The zebrafish motility mutant twitch once reveals new roles for rapsyn in synaptic function

Fumihito Ono, Anatoly Shcherbatko, Shin ichi Higashijima, Gail Mandel, Paul Brehm

Research output: Contribution to journalArticle

60 Scopus citations

Abstract

Upon touch, twitch once zebrafish respond with one or two swimming strokes instead of typical full-blown escapes. This use-dependent fatigue is shown to be a consequence of a mutation in the tetratricopeptide domain of muscle rapsyn, inhibiting formation of subsynaptic acetylcholine receptor clusters. Physiological analysis indicates that reduced synaptic strength, attributable to loss of receptors, is augmented by a potent postsynaptic depression not seen at normal neuromuscular junctions. The synergism between these two physiological processes is causal to the use-dependent muscle fatigue. These findings offer insights into the physiological basis of human myasthenic syndrome and reveal the first demonstration of a role for rapsyn in regulating synaptic function.

Original languageEnglish (US)
Pages (from-to)6491-6498
Number of pages8
JournalJournal of Neuroscience
Volume22
Issue number15
StatePublished - Aug 1 2002

Keywords

  • Muscle fatigue
  • Myasthenia gravis
  • Rapsyn
  • Synapse development
  • Synaptic depression
  • Tetratricopeptide repeats

ASJC Scopus subject areas

  • Neuroscience(all)

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